Trikalinos T A, Karvouni A, Zintzaras E, Ylisaukko-oja T, Peltonen L, Järvelä I, Ioannidis J P A
Clinical and Molecular Epidemiology Unit, Department of Hygiene and Epidemiology, University of Ioannina School of Medicine, Ioannina 45110, Greece.
Mol Psychiatry. 2006 Jan;11(1):29-36. doi: 10.1038/sj.mp.4001750.
Autism and autism-spectrum disorders exhibit high heritability, although specific susceptibility genes still remain largely elusive. We performed a heterogeneity-based genome search meta-analysis (HEGESMA) of nine genome scans on autism or autism-spectrum disorders. Each genome scan was separated in 30 cM bins and the maximum linkage statistic from each bin was ranked. Significance for each bin's average rank and for between-scan heterogeneity (dis-similarity in the average ranks) was obtained through Monte Carlo tests. For autism, data from 771 affected sibpairs were synthesized across six separate genome scans. Region 7q22-q32 reached genome-wide significance both in weighted and unweighted analyses, with evidence for significantly low between-scan heterogeneity. The flanking chromosomal region 7q32-qter reached the less stringent threshold of suggestive significance, with no evidence for low between-scan heterogeneity. For autism-spectrum disorders (634 affected sibpairs from five separate scans), no chromosomal region reached genome-wide significance. However, suggestive significance was reached for the chromosomal regions 17p11.2-q12 and 10p12-q11.1 in weighted analyses. There was evidence for significantly high between-scan heterogeneity for the former region. The meta-analysis suggests that the 7q22-q32 region should be further scrutinized for autism susceptibility genes, while autism-spectrum disorders seem to have quite diverse linkage signals across scans, possibly suggesting genetic heterogeneity across subsyndromes and subpopulations.
自闭症及自闭症谱系障碍具有很高的遗传度,尽管具体的易感基因仍大多未知。我们对九项关于自闭症或自闭症谱系障碍的全基因组扫描进行了基于异质性的基因组搜索荟萃分析(HEGESMA)。每次全基因组扫描被分成30厘摩(cM)的区间,并对每个区间的最大连锁统计量进行排序。通过蒙特卡洛检验获得每个区间平均排名以及扫描间异质性(平均排名的差异)的显著性。对于自闭症,来自771对患病同胞对的数据在六项独立的全基因组扫描中进行了整合。在加权和非加权分析中,7q22 - q32区域均达到全基因组显著性,且有证据表明扫描间异质性显著较低。侧翼染色体区域7q32 - qter达到了提示性显著性的较宽松阈值,没有证据表明扫描间异质性较低。对于自闭症谱系障碍(来自五项独立扫描的634对患病同胞对),没有染色体区域达到全基因组显著性。然而,在加权分析中,染色体区域17p11.2 - q12和l0p12 - q11.1达到了提示性显著性。对于前一个区域,有证据表明扫描间异质性显著较高。荟萃分析表明,7q22 - q32区域应进一步仔细研究自闭症易感基因,而自闭症谱系障碍在各次扫描中似乎有相当多样的连锁信号,这可能表明各亚综合征和亚人群之间存在遗传异质性。